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• W2058476475 abstract "Anomalous left coronary artery from the right pulmonary artery (RPA) with aortic fusion is a rare variant of anomalous left coronary artery from the pulmonary artery (ALCAPA). Three consecutive cases of this ALCAPA variant were managed, including a case with associated pink tetralogy. The challenges that can present in the diagnosis and treatment of this rare condition are described. A 3-month-old male infant was referred for elective repair of tetralogy of Fallot (TOF) with pulmonary stenosis. Echocardiography was consistent with a clinical picture of a “pink” TOF with normal coronaries. A total subannular repair of TOF with pulmonary stenosis was performed on cardiopulmonary bypass. During weaning, high left atrial pressures were noted with depressed left ventricular function. A transesophageal echocardiogram demonstrated satisfactory anatomic repair. Coronary anatomy was specifically examined. The left coronary artery appeared to originate from the left coronary sinus by both echocardiography and direct inspection. The patient was placed on extracorporeal membrane oxygenation (ECMO). Catheterization on ECMO the next day demonstrated an anomalous left main coronary artery (LMCA) arising from the RPA (Figure 1). At reoperation, careful dissection of the LMCA confirmed its origin from the RPA very close to the aorta. The ostium of the LMCA was excised as a button, but the vessel 3 or 4 mm distally could not be separated from the aortic wall to which it was fused. No intramural segment could be detected through an aortic incision and by probing the coronary artery. The short length of the coronary artery between the RPA and the point of fusion to the aorta did not allow direct transfer of the coronary button without risk of kinking. Thus, it was sutured to the lateral edge of an opening made in the ascending aorta with pericardium used to roof the opening (Figure 1). A femoral vein homograft segment was interposed between the distal pulmonary bifurcation and the proximal divided main pulmonary artery to prevent tension over the reimplanted coronary artery. Postoperative transesophageal echocardiogram showed good flow in the LMCA. The patient was weaned from ECMO 5 days later. Follow-up catheterization after 1 month revealed obstruction of the LMCA at the site of aortic anastomosis. This may be a consequence of late revascularization or kinking. The patient had a complicated and extended hospitalization, but he was eventually discharged. He died suddenly at home a few months later. The same condition was encountered at Children’s National Medical Center in 2 other male infants aged 4 and 6 months over a 6-month period. At operation, the left coronary artery had the same aortic fusion with RPA origin as previously mentioned. There was no evidence of dimpling within the left coronary sinus to attempt any fenestration. Given the short extramural course before fusion with the aorta, we transected the LMCA at its pulmonary end, which was oversewn. The LMCA was then filleted open to its aortic attachment, where a small intramural segment measuring less than 0.5 mm was observed. The filleted left coronary artery was directly anastomosed to the leftward and posterior aspect of the aorta. Postprocedure transesophageal echocardiogram in both patients demonstrated good flow in the LMCA. Both these patients did well in the postoperative period. Echocardiography at follow-up more than 1 year later revealed significant improvement in left ventricular systolic function and antegrade flow in the LMCA. Anomalous left coronary artery from the RPA with aortic trajectory and fusion was first described by Atik and colleagues1Atik E. Barbero-Marcial M. Ikari N.M. Vargas H. Albuquerque A.M. Kajita L. et al.Anomalia isolada da artéria coronária esquerda. Trajeto inusitado dentro de parede da aorta ascendente e inseção na artéria pulmonar direita. Relato de caso [Isolated anomaly of the left coronary artery: unusual origin within the ascending aortic wall and intersection in the right pulmonary artery. A case report].Arq Bras Cardiol. 1988; 51: 335-339PubMed Google Scholar in 1988. Because there was also a well-defined intramural course in Atik and colleagues’ cases and treatment was easily accomplished by fenestration, they termed this “a difficult condition to diagnose but easy to correct.” A thorough review of the literature reveals only 9 additional previous reports of such a variant2Atik E. Barbero-Marcial M. Tanamati C. Kajita L. Ebaid M. Jatene A. Anomalous origin of the left coronary artery from the right pulmonary artery with intramural aortic trajectory. Clinicosurgical diagnostic implications.Arq Bras Cardiol. 1999; 73: 181-190PubMed Google Scholar, 3Turley K. Szarnicki R.J. Flachsbart K.D. Richter R.C. Popper R.W. Tarnoff H. Aortic implantation is possible in all cases of anomalous origin of the left coronary artery from the pulmonary artery.Ann Thorac Surg. 1995; 60: 84-89PubMed Scopus (110) Google Scholar, 4Goldberg S.P. Mitchell M.B. Campbell D.N. Tissot C. Lacour-Gayet F. Anomalous left coronary artery from the pulmonary artery with an intramural course within the aortic wall: report of 3 surgical cases.J Thorac Cardiovasc Surg. 2008; 135: 696-698Abstract Full Text Full Text PDF PubMed Scopus (6) Google Scholar, 5Adachi I. Kagisaki K. Yagihara T. Hagino I. Ishizaka T. Kobayashi J. et al.Unroofing aortic intramural left coronary artery arising from right pulmonary artery.Ann Thorac Surg. 2008; 85: 675-677Abstract Full Text Full Text PDF PubMed Scopus (11) Google Scholar (Table 1). Although the diagnosis of ALCAPA was made in 11 of the 12 cases, the peculiar aortic trajectory of this variant was reliably predicted in only 1 case.5Adachi I. Kagisaki K. Yagihara T. Hagino I. Ishizaka T. Kobayashi J. et al.Unroofing aortic intramural left coronary artery arising from right pulmonary artery.Ann Thorac Surg. 2008; 85: 675-677Abstract Full Text Full Text PDF PubMed Scopus (11) Google Scholar The close approximation of the LMCA to the left aortic sinus, its vertical course running parallel to the ascending aorta, and the drop-out of the thin wall separating it from the aorta make preoperative characterization of the trajectory difficult by echocardiography.2Atik E. Barbero-Marcial M. Tanamati C. Kajita L. Ebaid M. Jatene A. Anomalous origin of the left coronary artery from the right pulmonary artery with intramural aortic trajectory. Clinicosurgical diagnostic implications.Arq Bras Cardiol. 1999; 73: 181-190PubMed Google Scholar, 6Barbero-Marcial M. Tanamati C. Atik E. Ebaid M. Jatene A. Anomalous origin of the left coronary artery from the pulmonary artery with intramural aortic route: diagnosis and surgical treatment.J Thorac Cardiovasc Surg. 1999; 117: 823-825Abstract Full Text Full Text PDF PubMed Scopus (18) Google Scholar Barbero-Marcial and colleagues6Barbero-Marcial M. Tanamati C. Atik E. Ebaid M. Jatene A. Anomalous origin of the left coronary artery from the pulmonary artery with intramural aortic route: diagnosis and surgical treatment.J Thorac Cardiovasc Surg. 1999; 117: 823-825Abstract Full Text Full Text PDF PubMed Scopus (18) Google Scholar defined the angiographic criteria for the diagnosis of this condition. In the setting of a significant left to right shunt and elevated pulmonary artery pressures, as was the case in our first patient, the diagnosis becomes even more difficult because there is little or no reversal of flow in the left coronary artery. At operation too, it is as difficult to diagnose this condition by inspection because the LMCA appears to arise normally from the left sinus of the aorta. A preoperative magnetic resonance angiography would be useful in the diagnosis of this rare condition whenever it is suspected. In all previous reports, surgical correction was achieved by unroofing the intramural segment alone in patients with a sufficient length of vessel that was intramural to allow this (Table 1). Patients who underwent unroofing alone or as part of a procedure in addition to direct reimplantation survived. Both the deaths reported previously occurred in patients who had coronary transfer alone. The principle to bear in mind at operation is that the coronary artery should not be allowed to kink during reimplantation. If there is a well-defined intramural segment, then unroofing is easy and the best option. If the intramural segment is small, then unroofing the small segment, filleting the coronary artery, and anastomosing to the aorta as described in our other 2 patients could be used with good results.Table 1Previous reports of anomalous left coronary artery from the right pulmonary artery with aortic fusionAtik and colleagues2Atik E. Barbero-Marcial M. Tanamati C. Kajita L. Ebaid M. Jatene A. Anomalous origin of the left coronary artery from the right pulmonary artery with intramural aortic trajectory. Clinicosurgical diagnostic implications.Arq Bras Cardiol. 1999; 73: 181-190PubMed Google ScholarN = 4Turley and colleagues3Turley K. Szarnicki R.J. Flachsbart K.D. Richter R.C. Popper R.W. Tarnoff H. Aortic implantation is possible in all cases of anomalous origin of the left coronary artery from the pulmonary artery.Ann Thorac Surg. 1995; 60: 84-89PubMed Scopus (110) Google ScholarN = 1Goldberg and colleagues4Goldberg S.P. Mitchell M.B. Campbell D.N. Tissot C. Lacour-Gayet F. Anomalous left coronary artery from the pulmonary artery with an intramural course within the aortic wall: report of 3 surgical cases.J Thorac Cardiovasc Surg. 2008; 135: 696-698Abstract Full Text Full Text PDF PubMed Scopus (6) Google ScholarN = 3Adachi and colleagues5Adachi I. Kagisaki K. Yagihara T. Hagino I. Ishizaka T. Kobayashi J. et al.Unroofing aortic intramural left coronary artery arising from right pulmonary artery.Ann Thorac Surg. 2008; 85: 675-677Abstract Full Text Full Text PDF PubMed Scopus (11) Google ScholarN = 1Our seriesN = 3OriginRPARPARPARPARPAAssociated lesionsNoneNoneRestrictive VSDNoneTOFPreoperative diagnosis ALCAPA+++++ (2/3) Anomalous course–––+–Extramural course??ShortShortShortIntramural course+ (3/4)++ (small)++ (small)Absent in 1Surgical correctionUnroofing alone in 3Coronary reimplantation without Unroofing in 1Unroofing aloneUnroofing with direct reimplantation in 2Coronary reimplantation alone without unroofing in 1→ Revision of anastomosis with unroofingUnroofing aloneUnroofing with direct reimplantation in 2Coronary reimplantation with pericardial roof without any unroofingResults3 survived1 death in reimplantation aloneSurvived3 survivedSurvived2 survived1 death in reimplantation aloneVSD, Ventricular septal defect. Open table in a new tab VSD, Ventricular septal defect. Anomalous left coronary artery from the RPA with aortic fusion is difficult to diagnose and may be challenging to repair when there is minimal intramural course." @default.
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